Benign Inflammatory Disease Research Articles

Aberrantly glycosylated integrin α3β1 is a unique urinary biomarker for the diagnosis of bladder cancer.

Bladder cancer (BC) is the most common malignancy of the urinary tract. We developed a new and ELISA kit for detecting aberrantly glycosylated integrin α3β1 (AG31) in human urine. We analysed urine samples (n=408) of patients with BC, renal cell carcinoma (RCC), prostate cancer (PC), cystitis, nephritis, and prostatitis from two centres in China. The subjects in the validation groups (n=2317) were recruited from other centres in China between July 2012 and September 2013. Receiver operating characteristic (ROC) curves were used to determine diagnostic accuracy. AG31 levels in urine samples were significantly higher in patients with BC than in any of the control subjects. Moreover, elevated levels of AG31 in urine could distinguish BC from benign inflammatory diseases. Finally, the urinary AG31 test was much more sensitive and specific than the NMP22 test. Therefore, the urinary AG31 test will provide an ideal and assay for the detection of BCs.

Islet autotransplantation improves glycemic control in patients undergoing elective distal pancreatectomy for benign inflammatory disease.

Islet autotransplantation (IAT) is increasingly being performed to mitigate against the diabetic complications of pancreatic resection in patients with benign inflammatory pancreatic disorders; however, the glycemic benefit of IAT in patients undergoing partial pancreatic resection is not known. We aimed to determine whether IAT improved glycemic outcomes in patients undergoing distal pancreatectomy for benign inflammatory disease. We performed a multicenter, retrospective case-control study of patients who underwent distal pancreatic resection with IAT at two U S tertiary care centers. The primary outcome was the mean change in pre- vs post-operative HgA1c following transplant as well as the development of new post-operative diabetes. Nine patients requiring distal pancreatectomy for benign disease underwent IAT and were compared to 13 historical controls without IAT. Baseline characteristics were similar between groups. With a median follow-up of 22months, those who received an IAT had a smaller increase in their pre- vs post-operative HgA1c (0.42 vs 2.83, P=.004), and one case patient (14.3%) vs three control patients (23.1%) developed new post-operative diabetes (P=.581). We conclude that patients undergoing distal pancreatic resection for benign inflammatory disease should be considered for IAT, as long-term glycemic outcomes appear to be improved in those undergoing transplant.

Treatment of idiopathic granulomatous mastitis and factors related with disease recurrence

Background/aim Idiopathic granulomatous mastitis is a rare, benign inflammatory disease of breast. There is no general agreement on the appropriate treatment choice. The aim of the study was to investigate the immunosuppressive administer for idiopathic granulomatous mastitis and risk factors related with disease recurrence.Materials and methods The data of 53 patients with idiopathic granulomatous mastitis were evaluated for this cross-sectional retrospective study. Demographic features and clinical characteristics and course of the patients were obtained from file records. Results The mean age of the patients was 37.2 ± 6.6 years. Fifty-one of 53 patients received immunosuppressive treatment with or without surgery. Forty-seven (88.6%) of the patients received only immunosuppressive treatment without surgery, while 4 (7.54%) patients received immunosuppressive treatment after surgery. Forty-one (77.3%) of 47 patients who had no surgical resection received methotrexate as immunosuppressive treatment. The other 6 (11.3%) patients received azathioprine or corticosteroid treatment. Complete or partial remission was observed in 50 (98%) of 51 patients who received immunosuppressive treatment, while only 1 (2%) patient did not reach remission. No factors were found related with recurrence of disease. Conclusion Methotrexate seems to be efficient in the treatment of idiopathic granulomatous mastitis and provides drug-free remission.

IgG4/IgG RNA ratio does not accurately discriminate IgG4-related disease from pancreatobiliary cancer.

Background & AimsIgG4-related disease (IgG4-RD) of the biliary tract and pancreas is often difficult to distinguish from pancreatobiliary cancer. The blood IgG4/IgG RNA ratio has been reported to discriminate IgG4-RD from primary sclerosing cholangitis/pancreatobiliary cancer with high accuracy. This study aimed to prospectively assess the diagnostic accuracy of the blood IgG4/IgG RNA ratio for distinguishing IgG4-RD from cancer in patients with a suspected pancreatobiliary malignancy.MethodsIn this prospective, single center, observational study, patients presenting at a specialized multidisciplinary, hepato-pancreato-biliary clinic with suspicion of pancreatobiliary malignancy were included. The IgG4/IgG RNA ratio (threshold 5.0%) was determined by quantitative PCR in addition to standard diagnostic procedures. Clinical, biochemical, radiological, and histo-/cytopathological findings were analyzed. For the diagnosis of IgG4-RD, the HISORt criteria were used as a reference standard. Malignancy was defined by the presence of neoplastic tissue at histo-/cytopathological examination.ResultsOverall, 213 consecutive patients (mean age 68 years) with a suspected pancreatobiliary malignancy were analyzed, of whom 3 patients were diagnosed with IgG4-RD and 178 patients were diagnosed with malignancy (165 patients with primary pancreatobiliary malignancy). The IgG4/IgG RNA ratio was true positive in 3 patients and false positive in 87 (40.8%) patients. In 123 (57.7%) patients the test was true negative. The sensitivity of blood IgG4/IgG RNA ratio was 100%, the specificity 58.6%, the positive predictive value 3.3%.ConclusionIn the setting of a high a priori risk of malignancy, an elevated IgG4/IgG RNA ratio did not accurately discriminate pancreatobiliary cancer from IgG4-RD as illustrated by low specificity and concordant low positive predictive value. We advise against the use of this test to discriminate IgG4-RD from pancreatobiliary malignancies.Lay summaryIgG4-related disease is a benign inflammatory multiorgan disease which predominantly affects the pancreas and biliary tree. Clinical symptoms, laboratory and imaging finding are often difficult to distinguish from pancreatic or biliary tract cancer. This prospective trial indicates that the recently proposed blood IgG4/IgG RNA ratio does not accurately distinguish benign IgG4-RD from malignant pancreatobiliary disease.

New Findings on Autoimmune Etiology of Idiopathic Granulomatous Mastitis: Serum IL-17, IL-22 and IL-23 Levels of Patients

Background Idiopathic Granulomatous Mastitis (IGM) is a benign chronic inflammatory breast disease that mimics breast cancer, and the etiopathogenesis has not yet been fully evaluated. Autoimmunity has received the most focus as a possible etiology. Our aim in this prospective clinical study was to investigate the possible association between the cytokines, interleukin IL-17, IL-22, IL-23 and IGM. Materials and Methods The current study was conducted in 26 women with histopathologically diagnosed IGM, and 15 control women of reproductive age having no breast disease history. Blood samples were collected, and serum concentrations of IL-17, IL-22, and IL-23 were determined. Results In the analysis of variables, the patients with IGM and the control group had statistically significant differences between serum IL-22 titers (p = 0.0378) and IL-23 titers (p = 0.0469. No statistically significant difference was found between IGM patients and the control group in serum IL-17 titers (p = 0.9724). Conclusion The results of the current study, especially pertaining to serum IL-22 and IL-23 levels, support the etiopathogenesis of IGM in favor of the autoinflammatory thesis. Nevertheless, this thesis should be supported by a large case number and prospective clinical studies.

A rare case of posttraumatic bilateral orbital myositis in a young boy — a case report

Orbital myositis (OM) is a benign inflammatory disease of the orbit characterised by a polymorphous lymphoid infiltrate with varying degrees of fibrosis, without a known local or systemic cause. In this paper, we present a case of a young boy who sustained a trauma to his eyes a few days prior to admission, after which he developed bilateral orbital pain and ocular motility limitation. He underwent the appropriate investigations including orbital imaging and blood laboratory workup, which were all consistent with a diagnosis of posttraumatic bilateral orbital myositis. He was treated with steroids for few weeks, and when the dose of steroids was tapered, he had a relapse of the same disease with a different presentation, which was later controlled with a higher dose of steroids, after which the patient went into remission.

Eosinophilic gastroenteritis presenting as persistent abdominal pain and ascites: A case report

Eosinophilic gastroenteritis (EGE) is a rare benign inflammatory disease affecting the infant and adult population. It presents by eosinophilic infiltration of the intestinal tract wall including the stomach, small intestines, and rarely the colon. EGE is divided into three subtypes depending on the layers and extent of bowel involved (mucosal, muscular, and serosal). Accurate diagnosis is still considered a challenge to professionals because of the protean manifestations; it needs a combination of clinical, laboratory, radiological, and histological workup. We report here a case of a 35-year-old male with a 3-month history of abdominal pain and distension. On examination, we found mild-to-moderate ascites and peripheral eosinophilia with markedly elevated eosinophils (85%). The imaging studies showed bowel wall thickening involving the entire small bowel and mild duodenitis. The diagnosis was confirmed by duodenal biopsy. The patient was managed with steroids (prednisolone 40 mg) and had an excellent response to the treatment. There is no single laboratory test or procedure to diagnose EGE; it requires a combination of laboratory, radiologic, and endoscopic findings. This report will guide decision-making for the initial phase of treatment and maintenance therapy in a more consistent way.

Therapeutic and Diagnostic Challenge of Idiopathic Granulomatous Mastitis: A Case Report and Review of the Literature

Idiopathic Granulomatous mastitis (IGM) is a chronic benign inflammatory disease of the breast that may mimic breast cancer. It is most common in parous young fertile women, although it can occur in nulliparous. It affects mostly women of childbearing age with a history of breastfeeding. IGM is an idiopathic disease due to the influence of some environmental factors in genetically predisposed subjects. Several pathogenic hypothesis have been proposed in the last years (autoimmune, hormonal, infective genesis). Because the clinical presentation can mimic infectious mastitis or inflammatory carcinoma, the disease course is often protracted. The diagnosis is made by histopathology. Ultrasound, mammography, and magnetic resonance imaging are not specific; however, ultrasound and mammography should be done to exclude other pathologies. There is no universal treatment of GM. We report in this manuscript one case of IGM through which we mention a therapeutic and diagnostic challenge with a brief review of the literature. The management was successful despite the long way made by the patient before she acceded to a proper treatment.

AYURVEDIC MANAGEMENT OF IDIOPATHIC GRANULOMATOUS MASTITIS (STHANAVIDRADHI): A CASE STUDY

Idiopathic Granulomatous Mastitis (IGM) is described as an uncommon, benign inflammatory breast disease of unknown etiology. Although its exact prevalence is unknown, IGM is considered to be very rare. Here, we report the case of a 36-year-old female who came to our hospital with complaints of pain and lump in the right breast since 2 months. We diagnosed the case as Sthana Vidradhi and vathakaphasamana chikithsa was administered. The wound was managed with vranaropakalepa and kashaya. After treatment, the wound was healed. Pain and tenderness were reduced and the lump was disappeared after two months of medication. The medication continued and complete recovery was noted after 6 months of treatment. The patient was monitored for six months after the complete cure.&nbsp

CARCINOMA CUNICULATUM OF THE ALVEOLAR RIDGE

Carcinoma cuniculatum (CC) is a rare, well-differentiated variant of oral squamous cell carcinoma that usually affects the mucoperiosteum of older patients. Oral CC appears mainly as locally destructive tumors with superficial drainage of keratin, with low risk of metastasis. A 56-year-old white woman, a nonsmoker and nondrinker, presented a nonhealing gingival ulcer with duration of 2 months. Intraoral examination showed a well-defined irregular ulcer in the posterior upper alveolar ridge with drainage of a whitish material suggestive of keratin during palpation. Periapical radiograph showed an ill-defined osteolytic lesion. Microscopic evaluation showed well-differentiated proliferation of squamous epithelium with mild cytologic atypia exhibiting multiple infiltrative and branching keratin-filled epithelial crypts. Oral CC may appear as an irregular ulcer in the alveolar ridge with drainage of whitish material, which may be clinically similar to benign inflammatory diseases of the jaws. Carcinoma cuniculatum (CC) is a rare, well-differentiated variant of oral squamous cell carcinoma that usually affects the mucoperiosteum of older patients. Oral CC appears mainly as locally destructive tumors with superficial drainage of keratin, with low risk of metastasis. A 56-year-old white woman, a nonsmoker and nondrinker, presented a nonhealing gingival ulcer with duration of 2 months. Intraoral examination showed a well-defined irregular ulcer in the posterior upper alveolar ridge with drainage of a whitish material suggestive of keratin during palpation. Periapical radiograph showed an ill-defined osteolytic lesion. Microscopic evaluation showed well-differentiated proliferation of squamous epithelium with mild cytologic atypia exhibiting multiple infiltrative and branching keratin-filled epithelial crypts. Oral CC may appear as an irregular ulcer in the alveolar ridge with drainage of whitish material, which may be clinically similar to benign inflammatory diseases of the jaws.

Idiopathic granulomatous mastitis: a diagnostic challenge

Introduction: Idiopathic granulomatous mastitis is an uncommon and benign inflammatory breast disease that usually occurs in young women of childbearing age. Most common presentation is a slowly growing breast mass that is hard in consistency and can greatly mimic breast cancer. Mammographic and ultra-sonographic findings are non- specific and usually mimic malignant masses leading to higher BIRADS grading in reports. The only way to reach a definitive diagnosis is by histopathological diagnosis. Material & Methods: In the present study eight patients were presented to The Integrated Breast Care Centre (IBCC) department of AIIMS Rishikesh with breast lump or nipple discharge during January 2019 to June 2019. All the patients underwent mammography and ultrasonography followed by FNAC or Biopsy of the lesion as findings were mimicking malignant etiology. Results: On HPE all the patients showed features of granulomatous inflammation and diagnosis of idiopathic granulomatous mastitis was made. TB was excluded by CBNATT (Cartridge Based Nucleic Acid Amplification Test) examination of the aspiration cytology samples. To our knowledge, such a report is the first of its kind to address this topic in this region. Conclusion: In a young child-bearing age group female presenting with breast mass a differential of idiopathic granulomatous mastitis should also be considered. The clinicians as well as radiologists should be aware of this entity so that appropriate management may be done.

Surgical treatment of granulomatous mastitis: Our experience in Bahrain.

Granulomatous mastitis (GM) is a rare, benign inflammatory disease of the breast with unknown etiology. There is no universally accepted treatment for GM. The aim of this study was to show our experience with surgical treatment of GM. A retrospective review was performed for 29 cases that were surgically treated at Salmaniya Medical Complex (SMC) in Bahrain between 2010 and 2017. All patients underwent surgical excision with debridement and removal of retroareolar ductal system. None of these patients experienced recurrence in the follow-up period. Complete surgical excision of the whole inflammatory mass is the treatment of choice.

Imaging features of granulomatous mastitis in 36 patients with new sonographic signs.

Idiopathic granulomatous mastitis is a rare relapsing benign inflammatory breast disease with unknown etiology. Its clinical features and imaging signs may mimic inflammatory breast cancer or some other inflammatory breast disease. This may interfere with correct and timely diagnosis and thus impose an additional burden on the costs of diagnosis and therapy, as well as patient anxiety. We aimed to characterize the imaging findings of this disease and introduce two new imaging signs. This prospective study examined 36 patients with imaging and a clinical diagnosis of mastitis granulomatosis who were untreated and then confirmed by pathology. Demographic information, clinical data, imaging findings, and signs were recorded. The age range of the patients was 22-60years with an average of 36years. Most of the patients (78%) were at reproductive age. None of the patients had a family history of granulomatous mastitis. Most patients with granulomatous mastitis (89%) lived in regions with low socioeconomic status. For most patients, sonography indicated a heterogeneous hypoechoic mass with irregular shape and ill-defined margin (26 cases; 72.2%). Focal asymmetry (36%) and obscured mass (36%) were the most common mammographic findings. Two signs of duct ectasia containing secretion and high-flow pseudocyst appearance were described. Mammographic and ultrasound findings can highly suggest a diagnosis of granulomatous mastitis in an appropriate clinical context.

Manage endometriosis-related pain with hormonal suppression therapies

Endometriosis is a benign chronic inflammatory disease that mostly affects women of reproductive age. The disease is often associated with dysmenorrhoea, pelvic and non-menstrual pain, and infertility. NSAIDs and hormonal suppression therapies (i.e. estroprogestins and progestins) are generally used as first-line treatment to alleviate pain symptoms. Second-line options include other hormonal suppressants, such as gonadotropin-releasing hormone agonists and antagonists and, less commonly, danazol and aromatase inhibitors.

Delayed breast reconstruction in idiopathic granulomatous mastitis

Idiopathic granulomatous mastitis is an exceptionally rare, chronic benign inflammatory disease of the breast, more common in developing countries and infrequently seen bilaterally. The symptoms can mimic many other breast conditions, including inflammatory breast carcinoma, and the diagnosis is one of exclusion. The management includes non-surgical treatments, such as broad spectrum antibiotics, steroids and methotrexate, and surgical options, comprising of wide local excision and even simple mastectomy. The case notes of patients with idiopathic granulomatous mastitis referred to our Breast Unit were reviewed and data was collected on patients demographics, medical and surgical treatments and subsequent outcomes. Four patients were treated in our unit, all Caucasian women, three of whom were bilateral cases. Three of the patients were reconstructed using oncoplastic techniques whilst the fourth patient awaits reonstruction. Oncoplastic breast reconstructive techniques play a fundamental role in the management of these patients, as they give the surgeon the freedom to perform a wider resection, thus reducing the rate of recurrence, whilst at the same time, knowing that there are reconstructive options to restore any breast aesthetics. As such, the value of a multidisciplinary team cannot be underestimated. Level of Evidence: Level V, therapeutic study.

The role of biologics in the treatment of chronic granuloma annulare.

Granuloma annulare (GA), a benign inflammatory skin disease, is considered a Th1-type delayed hypersensitivity reaction. Localized GA is likely to resolve spontaneously, whereas disseminated GA (DGA) may persist for decades and can be resistant to treatment. Biologics including TNF-α inhibitors have been proposed and utilized as salvage therapy for GA and other related diseases, interstitial granulomatous dermatitis (IGD), and actinic granuloma (AG). A systematic review was conducted using the combination of search terms "granuloma annulare," "interstitial granulomatous dermatitis," or "actinic granuloma" and either "biologics," "etanercept," "adalimumab," "infliximab," "ustekinumab," "ixekizumab," "secukinumab," "guselkumab," "golimumab," "brodalumab," "tildrakizumab," or "certolizumab" from the years 1970-2017. Review of the literature revealed that 79.3% of the patients with GA, IGD, or AG who had been treated with demonstrated TNF-α inhibitor therapy a clinical response. TNF-α inhibitor therapy has been used to treat chronic GA, IGD, and AG that involved extensive body surface areas. However, the literature is limited to case series lacking control groups. Randomized, controlled trials are required to establish evidence-based treatment of GA and related cutaneous, granulomatous conditions.

Abdominal wall endometriosis – clinical pattern and treatment

Endometriosis is a benign inflammatory disease that affects a large proportion of women in their reproductive age. A peculiar type is the one situated on the postsurgical scars in the form of endometriomas. Whilst most patients with this pathology do not have a history of pelvic endometriosis, the explanation for its appearance is the theory of implantation of endometriotic cells during surgery, as well as the existence of primitive, pluripotent mesenchymal cells that have undergone differentiation and metaplasia processes. Pain is the main symptom of this condition, accompanied frequently by the appearance of an abdominal wall mass, which are both influenced by menstrual periods. Endometriosis is an estrogen dependent disease, thus the medical treatment could comprise contraceptives containing progesterone, as well as antiestrogens, but the long-term success rate is low, the treatment of choice being the surgical one.

Diagnostic techniques and multidisciplinary approach in idiopathic granulomatous mastitis: a revision of the literature.

Idiopathic granulomatous mastitis (IGM) is a chronic benign inflammatory disease of the breast that may mimic breast cancer. It is most common in parous young fertile women, although it can occur in nulliparous women and in men. IGM is an idiopathic disease due to the influence of some environmental factors in genetically predisposed subjects. Several pathogenic hypothesis have been proposed in the last years (autoimmune, hormonal, infective genesis). IGM presents as a painful palpable mass located in one of the two udders. The skin is usually normal but could present signs of inflammation with or without lymph nodes involvement. Ultrasonography, mammography, magnetic resonance can be diagnosed an IGM, but pathognomonic radiological signs has not yet reported in literature. Biopsy findings show granulomatous lesion centered on the breast lobule, as in granulomatous mastitis induced by tuberculosis or sarcoidosis. The aim of this review of literature is to verify the development of new advanced diagnostic techniques and multidisciplinary approach for this condition. In the last years innovative approaches have modified IGM diagnosis and therapy, avoiding surgery in most of cases, introducing a more conservative medical approach based on recent etiopathological hypothesis. (www.actabiomedica.it)

The Effect of Nasal Steroid Spray on Contact Granuloma of Larynx

Background and Objectives Laryngeal contact granuloma is benign inflammatory disease induced by excessive mechanical contact of larynx such as endotracheal intubation, voice abuse, laryngeal microsurgery as well as laryngopharygeal reflux. Because it is caused by various risk factors, multiple treatment modalities are required. The purpose of study is to evaluate treatment effect of topical steroid through nasal cavity in contact granuloma. Materials and Method Fifty-two patients were enrolled in this study with exception of intubation granuloma. Patients were classified with four groups (Proton pump inhibitor (PPI), Nasal steroid spray (SPR), PPI+SPR, Observation) according to treatment modality. Results Patients who treated with PPI (Odds ratio 2.45, p=0.03) and combination of PPI and SPR (Odds ratio 2.88, p<0.01) had significantly better response than patients who not treated with medical therapy. Conclusion Combination therapy of nasal steroid spray and PPI is effective for contact granuloma of larynx and considered as a treatment of choice rather than PPI only treatment. Key words: Contact granuloma ㆍ Larynx ㆍ Nasal steroid spray 핵심용어: ì ‘ì´‰ì„± ìœ¡ì•„ì¢ ã† 후두 ㆍ 비강 ìŠ¤í Œë¡œì´ë“œ 분무

Clinical and Radiological Differences Between Fistular and Non-Fistular Idiopathic Granulomatous Mastitis

Background: Idiopathic granulomatous mastitis (IGM) is a rare and benign chronic inflammatory breast disease. The disease is usually associated with recurrent attacks and may result in the formation of fistulas. Fistula or ulceration on the skin of the breast makes it difficult to medication treatment and may necessitate extensive skin excision in patients undergoing surgery. Objectives: This study aimed to compare the clinical and radiological findings between fistular and non-fistular idiopathic granulomatous mastitis. Patients and Methods: The clinical and radiologic findings of 52 women with a histopathology diagnosis of idiopathic granulomatous mastitis were retrospectively evaluated. All the patients were assessed using ultrasonography and magnetic resonance imaging. Of the 52 patients, 20 underwent mammography. The findings in patients with fistular and non-fistular idiopathic granulomatous mastitis were compared. Results: Erythema, ulceration, and nipple changes were more common in patients with fistular idiopathic granulomatous mastitis. While the most common finding in ultrasounography in fistular IGM patients were collection areas with complicated cysts consistent with abscess, the finding in non-fistular IGM patients were multiple irregularly shaped hypoechoic masses. In magnetic resonance imaging, the most common finding in fistular IGM patients was non-mass lesions with regional clustering ring enhancement, whereas the most frequent finding in non-fistular IGM patients was non-mass lesions with regional heterogeneous enhancement. There were statistically significant differences between findings of both groups. Conclusion: There are differences in the clinical and radiologic findings of fistular and non-fistular idiopathic granulomatous mastitis. Ultrasonography and magnetic resonance imaging could be useful in identifying patients with a high risk of developing fistulas.